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Case Report

Primary oral tuberculosis: A case series of a rare disease


Rizwan Hamid1, Prenika Sharma1, Rayees Ahmad Sheikh1, Muzafar Bhat2
Departments of 1Oral Medicine and Radiology and 2Periodontics, GDC, Srinagar, Jammu and Kashmir, India

Abstract Tuberculosis (TB) is still one of the most life‑threatening infectious diseases, resulting in high mortality in
adults and is commonly found in developing countries. Lung is primarily affected while extrapulmonary
TB is rarely encountered. Oral lesions, although rare, can be seen in both primary and secondary stages
of TB. Primary oral TB may present a diagnostic challenge as its clinical features can be nonspecific that
mimics other diseases and is usually misdiagnosed. Thus, it is very important to be aware and be highly
suspicious of oral TB especially in endemic area. We share 4 such cases of primary oral TB with uncommon
presentations (two on the gingiva, one on the palate and one on the tongue) The diagnosis was made by
histopathological examination, polymerase chain reaction analysis and Mantoux test. They were successfully
treated with antituberculous treatment. In secondary TB, the oral manifestations may be accompanied
by lesions in the lungs, lymph nodes or in any other part of the body and can be detected by a systemic
examination.

Keywords: Gingiva, oral tuberculosis, palate, primary, tongue, tuberculosis

Address for correspondence: Dr. Rizwan Hamid, Department of Oral Medicine and Radiology, GDC, Srinagar, Jammu and Kashmir, India.
E‑mail: drrizwanhamid@gmail.com
Submitted: 01-Jun-2019, Revised: 04-Mar-2020, Accepted: 23-Mar-2020, Published: 09-Sep-2020

INTRODUCTION is usually presented as an ulcer. It has been hypothesized


that auto‑inoculation may occur when the infected
Tuberculosis (TB) is still among the most life‑threatening pulmonary mucus interacts with wounded, susceptible
infectious diseases, resulting in high mortality in adults.[1] areas of the mucosa, eliciting the emergence of lesion.[5]
It is a chronic infectious granulomatous disease caused by So far, cases of primary TB of the tongue are published
Mycobacterium tuberculosis, an acid‑fast bacillus (AFB), and as an anecdotal case reports because of extreme rarity.[6]
less frequently by ingesting un‑pasteurized cow’s milk that Tubercular foci in OC secondary to primary TB of the lung
is infected by Mycobacterium Bovis or by other atypical is again uncommon if not rare.[7] The incidence of oral
mycobacteria.[1] Both primary and secondary types of TB TB is only 0.1%–0.4%.[8] TB of OC even as a secondary
can cause lesions in the oral cavity (OC).[2] In secondary form is uncommon.[7] When oral lesions of TB are the
TB, lesions of the OC may accompany lesions in the sole manifestations of the disease, the clinician may face
pharynx, lungs, lymph nodes or skin.[2] Extrapulmonary a diagnostic challenge.
TB accounts for 25% of the cases with 10%–35% detected
in the head and neck region.[3,4] Oral manifestation of TB The World Health Organization  (WHO) definition
may affect people of all ages, especially the elderly, and of the case of extrapulmonary TB (EPTB) is: “any

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DOI: How to cite this article: Hamid R, Sharma P, Sheikh RA, Bhat M. Primary
10.4103/jomfp.JOMFP_174_19 oral tuberculosis: A case series of a rare disease. J Oral Maxillofac Pathol
2020;24:332-8.

332 © 2020 Journal of Oral and Maxillofacial Pathology | Published by Wolters Kluwer - Medknow
Hamid, et al.: Primary oral tuberculosis – A case series of a rare disease

bacteriologically confirmed or clinically diagnosed case of palpation, the swelling was slightly tender, firm and had
TB involving organs other than the lungs, e.g., pleura, lymph a tendency for bleeding on provocation. The rest of
nodes, abdomen, genitourinary tract, skin, joints and bones, the OC was normal. Complete hemogram and IOPA
meninges.” From this definition are excluded the cases with radiograph of 11, 12, 21 and 22 were advised. Results
both pulmonary TB and EPTB, which are recommended were within normal limits, except for a marginal rise in
to be classified as pulmonary TB cases.[9,10] The lack of leukocyte count and an elevated erythrocyte sedimentation
specific pathognomonic signs makes the diagnosis of rate (ESR). IOPA radiograph revealed interdental bone
EPTB to be often overlooked by the clinician.[2,11] Orofacial loss in relation to 11 and 21. The patient was then
TB is uncommon and presents at different sites such as advised tuberculin test, chest X‑ray and sputum culture.
the mandible (alveolar and basal bone), head, face and A tuberculin (Mantoux) test was positive, suggesting
neck lymph nodes, tonsils, salivary glands, maxilla and tubercular infection. Chest radiography (posteroanterior
maxillary antrum, hard palate and soft tissues such as soft view) revealed no abnormalities [Figure 1b]. Culture of
palate, uvula, the gingiva, tongue, muscles of mastication sputum was negative for M. tuberculosis. An incisional
and buccal mucosa.[11,12] OC involvement is very rare and biopsy from the maxillary labial gingiva adjacent to
was reported a long time ago. Morgagni described first case the central incisors was performed. Histopathological
of lingual TB in 1761.[6] De Paoli reported the first case examination revealed clusters of epithelioid cells,
of parotid gland TB in 1893.[13] The incidence of TB in caseating necrosis and numerous Langhans‑type giant
underdeveloped countries is increasing, and this is thought cells surrounded by a chronic inflammatory type of
to be because of associated poor hygiene conditions and infiltrate  [Figure  1c and d]. In view of these findings,
the greater prevalence of AIDS.[14] Here, we report four a working diagnosis of primary tuberculous gingival
cases of primary oral TB (two on the gingiva, one on the enlargement was made. The patient was later administered
palate and one on the tongue). Clinical features of cases antitubercular regime, and within 2 months of therapy, the
are summarized in Table 1. lesion healed spontaneously [Figure 1e]. No recurrence
was observed even after 6 months of follow‑up. During
CASE REPORTS this period, the patient was instructed not to undergo any
surgical procedure within the OC. Further, conservative
Case 1 periodontal therapy, which included scaling and root
A 37‑year‑old female reported to the department of planning, was carried out with minimal trauma to gingival
oral medicine and radiology with a chief complaint of and after consulting the physician in‑charge.
nonpainful swelling of the upper anterior gingiva for the
past 8 months. The gingiva increased gradually in size with Case 2
time. The patient had a history of weakness over the past A 45‑year‑old female presented with a chief complaint
3–4 months, loss of appetite and loss of weight of about of painful ulcer on the palate for the last 1 year, which
5 kg during the past 3 months. Her medical history revealed gradually increased to the present size. She had received
no systemic problems, no cough with expectoration, no antibiotics (amoxicillin plus clavulanate) prescribed by
known history of contact with a tuberculous patient and local practitioners, but there was no response. She did
no history of dental trauma or any surgery in the affected not have any systemic complaints such as cough, fever
area. On examination, she was of average built, pulse, or weight loss and had no history of any allergy. There
temperature and respiration rates were normal. Extraoral was no cervical lymphadenopathy or any other abnormal
examination revealed no cervical lymphadenopathy. findings. Intraoral examination revealed gingival ulcerations
Intraoral examination showed diffuse enlargement of involving marginal and attached gingiva in relation to
labial maxillary gingiva (marginal and attached gingiva) 11–14 and mucosa of the anterior hard palate. The ulcer
and alveolar mucosa extending from tooth number 13–23. is indurated, irregular, having an undermined margin and
The gingiva was red, irregular, pebbled and granular a yellowish granular necrotic base [Figure 2a]. Owing
in appearance with surface ulceration [Figure 1a]. On to suspicion of a malignant lesion, an incisional biopsy

Table 1: Demographic and clinical characteristics


Case number Age (year) Gender Presented as Masquerading
Case 1 37 Female Nonpainful swelling of the upper anterior gingiva NUG
Case 2 45 Female Painful ulcer on palate NUG/NUP
Case 3 23 Female Nonpainful swelling of the gingiva Gingival overgrowth
Case 4 49 Male Painful and non‑healing ulcerated lesion of the tongue Oral SCC
NUG: Necrotizing ulcerative gingivitis, NUP: Necrotizing ulcerative periodontitis, SCC: Squamous cell carcinoma

Journal of Oral and Maxillofacial Pathology | Volume 24 | Issue 2 | May-August 2020 333
Hamid, et al.: Primary oral tuberculosis – A case series of a rare disease

was undertaken from anterior mucosa of hard palate diseases for further management. Treatment was started
following the baseline investigations which were within with isoniazid  (300  mg/day), rifampicin  (600  mg/day),
normal limits. Histopathological examination (HPE) of pyrazinamide (1500 mg/day) and ethambutol (900 mg/day)
the surgical specimen showed a conserved epithelium for 2 months and the patient was asked to continue with
covering the subepithelial layers, with widespread caseating the first two drugs for the next 4 months. No pulmonary
granulomas surrounded by lymphocytes, epithelial cells and signs and symptoms were present. The oral lesions resolved
Langhans‑type giant cells [Figure 2b]. No neoplastic changes within 4 weeks of treatment [Figure 2c]. The patient
were observed. Ziehl–Neelsen staining was negative. With was followed for 9 months after the treatment with no
all these data, the diagnosis of oral TB was suggested recurrence of lesion.
and systemic analyses were performed to determine its
primary or secondary origin. The Mantoux test showed Case 3
a positive reaction. Chest X‑ray did not show any lesion A 23‑year‑old girl reported to the department of oral
suggestive of pulmonary TB. Three sputum specimens medicine and radiology with progressive, nonpainful
were smear negative and culture negative. Although the swelling of the gingiva on the labial aspect of the upper
first clinical impression raised suspicion of a malignant and lower anterior teeth with 6 months’ duration. There
or traumatic process, these were both dismissed based on was a history of evening rise in temperature and weakness
the pathology results. In fact, the presence of caseating over the last 3 months. The patient also had a loss of
granulomas surrounded by lymphocytes, epithelial cells and appetite over the last 4 months and a weight loss of about
Langhans‑type giant cells confirmed the diagnosis of TB. 4.5 kg during the last 8 months. There were no systemic
The patient was referred to the department of infectious problems, no cough with expectoration and no history of
dental trauma or surgery in the affected area. Extraoral
examination revealed no cervical lymphadenopathy.
Intraoral examination revealed diffuse enlargement of the
upper and lower gingiva on the labial surface of anterior
maxillary and mandibular teeth [Figure 3a]. On palpation,
the swelling was slightly tender and firm. The rest of the
OC was normal except for the few deep carious teeth.
Differential diagnoses were enlargement due to drugs,
a e
infection and hematologic malignancy. The possibility of
drug‑induced enlargement was ruled out based on medical
history. The biochemical tests were within normal limits,
except for a marginal rise in leukocyte count (13 × 10 9/L)
and an elevated ESR of 56 mm/h (Westergren method),
which ruled out leukemia‑associated enlargement and
b c dd raised the possibility of one of the common causes of high
Figure 1: (a) Red, irregular, pebbled and granular with surface ESR, TB. An incisional biopsy was carried out under LA in
ulceration (b) chest radiograph showing absence of foci of infection. relation to the gingiva of the mandibular right central incisor,
(c) Photomicrograph showing granulomatous inflammation with in collaboration with the department of periodontics and
Langhanæs giant cells and focal caseous necrosis in H&E (×20).
(d) Photomicrograph showing Langhanæs giant cells in H&E implantology. Histopathological examination was carried
stain (×40). (e) Healing lesion (2 months after start of ATT) out that revealed clusters of epithelioid cells surrounded

a b c
Figure 2: (a) Indurated irregular ulcer, having a yellowish granular necrotic base (b) histopathological slide showing giant cells (×40) (c) oral
lesions resolved within 4 weeks of treatment

334 Journal of Oral and Maxillofacial Pathology | Volume 24 | Issue 2 | May-August 2020
Hamid, et al.: Primary oral tuberculosis – A case series of a rare disease

a b c

d e f
Figure 3: (a) Diffuse enlargement of both the arches and lobulated appearance of the mandibular labial gingiva extending up to the left first
molar (b) microscopic picture reveals numerous non‑caseating granulomas (×10) (c) histopathological picture of the lesion under ×40 magnification
reveals Langerhans giant cells and epithelioid cells (d) chest X‑ray does not reveal any abnormality (e) immediate post‑operative picture of the
lesion (f) intraoral picture after 1‑year follow‑up does not reveal any recurrence of the lesion

by a chronic inflammatory type of infiltrate. There was no and salivary contamination. After completion of a 6‑month
evidence of caseating necrosis, but numerous Langhans regimen of basic periodontal therapy, which included
giant cells were visible in the clusters of epithelioid cells scaling and root planning, oral hygiene instructions were
suggestive of a “hard tubercle” [Figure 3b and c]. To instituted under CDC‑issued guidelines. This resulted in
eliminate the possibility of localized granulomatous changes significant regression of the enlarged gingivae in both the
superimposed on an area of gingival enlargement, incisional arches. Gingivectomy and gingivoplasty were performed to
biopsy was repeated in the remaining three quadrants. shape and contour the residual enlargement under universal
Histopathology showed similar granulomatous changes in aseptic conditions [Figure 3e]. No recurrence of lesion
all tissue specimens examined. The tuberculin (Mantoux) occurred during 1‑year follow‑up [Figure 3f].
test was positive, suggesting tubercular infection.
Case 4
Chest radiography (posteroanterior view) revealed no
A 49‑year‑old male was referred to our department with
abnormalities [Figure 3d]. A computed tomography scan of
the complaint of painful and non‑healing ulcer of the
the head and neck region was also performed to determine
tongue for 4 months. There was no history of fever, night
the status of the underlying maxilla and mandible. The
sweats, cough, decreased appetite and weight loss. He did
scan did not reveal any bone abnormalities. A culture of
not give any history of traumatic episode preceding the
the sputum, obtained by forceful coughing, was negative
development of tongue ulcer. He denied any history of
for M. tuberculosis. Special staining of formalin‑fixed,
similar lesions in the past. He is an occasional smoker. The
paraffin‑embedded tissue specimens for Mycobacteria, rest of his medical and surgical history was unremarkable.
i.e., Ziehl–Neelsen and auramine–rhodamine stain, was His dental history was not significant. There was no history
negative. An immunologic test to detect antibodies against of TB in his family members. He was repeatedly treated
Mycobacterium in the patient’s serum (ELISA) was in a government hospital with topical antiseptics and oral
positive. Polymerase chain reaction (PCR) assay was also antibiotics (ampicillin plus cloxacillin and metronidazole)
carried out using six 5‑µm sections of paraffin‑embedded and analgesics, but the lesion did not subside so he was
tissue to identify specific sequences of M. tuberculosis referred to our hospital for the further management. On
complex, with adequate controls. The DNA was used as general examination, he was of average built. Intraoral
an amplifying target for the sequence IS‑6110, which is examination revealed an indurated ulcer measuring
specific for M. tuberculosis. Positive PCR results confirmed 1.5 cm × 1.0 cm on the left dorsolateral border of anterior
the presence of M. tuberculosis in the tissue samples. In view part of tongue. It was tender on palpation with irregular
of these findings, a final diagnosis of primary tuberculous borders and did not bleed on touch. The remaining part of
gingival enlargement was made. In consultation with the the tongue appeared normal in texture and color. Mobility
patients’ physicians, antitubercular therapy (ATT) was of the tongue was normal. He had poor oral hygiene
initiated. During this period, the patient was instructed along with sharp tooth in relation to 34. There were no
not to undergo any ultrasonic scaling and polishing or palpable cervical lymph nodes. Provisional diagnosis of
surgical procedure within the OC and was warned about chronic traumatic ulcer was made as the possibility of
the chance of transmitting the disease to others via aerosol ulcer due to repeated trauma by the sharp tooth. Chest
Journal of Oral and Maxillofacial Pathology | Volume 24 | Issue 2 | May-August 2020 335
Hamid, et al.: Primary oral tuberculosis – A case series of a rare disease

X‑ray did not reveal any evidence of active lesion. Sputum of which affect the tongue.[22] Three of the cases in our
for AFB was negative on Ziehl–Neelsen stain. Complete series were chronic ulcers and one presented as gingival
blood count was within normal limit. His ESR value was overgrowth. The ulcers were on the lateral margin of
20 mm/hr. Serum biochemistry and renal function tests tongue, anterior gingiva and anterior palate. Primary TB
were within normal limits. Serological investigation for of the OC is extremely rare and the published literature
human immune deficiency virus (HIV) was negative. is only in the form of case reports.[6,23,24] There appears
Coronoplasty of adjacent sharp cusps was done, and since to be a geographical variation regarding which age group
the ulcer presented for more than 2 months, an incisional is affected by primary TB. In Western literature, it is
biopsy was taken under local anesthesia containing an mentioned that primarily oral TB affects children and
ulcerated lesion along with the normal looking margin adolescents and is often associated with enlarged cervical
and the specimen was sent for HPE. Microscopically, the lymph nodes.[25] A few case reports from India also fall into
lesion revealed the stratified squamous epithelium with younger age group.[6,24] In this series, the mean age of the
granulomatous inflammation containing Langhans‑type patients was 38.5 years, while youngest being 23‑year‑old
giant cells, epithelioid cells and foci of caseous necrosis, female. TB of OC can be seen in adults, but primary TB
strongly suggestive of TB. The patient was then started of lip and uvula among pediatric age group has been
on ATT. The ulcer gradually started fading once drug reported occasionally.[24] Wang et al.[26] reported a series of
therapy was started (ATT) and the tuberculous ulcer 20 cases from Taiwan, wherein 55% of the patients were
healed completely by the 2 months of ATT containing older than 50 years and the most common location was
isoniazid, rifampicin, pyrazinamide and ethambutol. The buccal mucosa and/or vestibule (5 cases), followed by the
patient was advised to continue ATT containing isoniazid alveolar mucosa (4), palate (2), lip (2) and tongue (1 case).
and rifampicin for another 4 months. At the end of total Rarely, direct inoculation may result in primary oral TB.
6 month’s ATT course, there was no evidence of ulcer at The site most commonly affected is the gingiva where
the primary site. primary TB appears as a diffuse erythematous patch or
as diffuse gingival enlargement.[27] Primary TB of the OC
DISCUSSION has been reported in HIV infected immune deficient cases
as an indicator of HIV infection; however, most of the
Primary TB (TB) of the OC, including tongue, is very published literature pertains to cases with intact immune
rare because of continuous cleaning of oral mucosa by status similar to the cases under discussion.[23,28] Other
saliva and paucity of lymphoid follicles in tongue.[15‑17] TB lesions, diffuse glossitis or fissures, have also been
Secondary TB of OC is 0.2%–1.5% of extrapulmonary reported, but they are exceedingly rare.[29] Although the
TB cases.[18,19] The WHO estimates that 2 billion people dorsal surface is more commonly involved, involvement
or one‑third of the world’s population are infected with of the ventral surface has also been described.[29]
tuberculous bacilli and the global TB incidence is growing
at 1% a year.[15] Despite these staggering figures, TB of The tuberculin sensitivity assay, also called Mantoux
the OC is rare.[15‑17] Furthermore, most cases of oral TB test, is the standard procedure to diagnose TB. The assay
are mainly secondary to pulmonary TB and rarely primary includes the intradermal inoculation of a purified protein
in origin.[15] Such lesions are suspected to be caused by derivative of M. tuberculosis to assess the cellular immune
implantation of infected sputum into a break in the response to the antigens. An inflammatory reaction takes
mucosal surface during coughing episodes. Transmission place in M. tuberculosis sensitized patients. Inspection is
during dental practice has also been described.[15] Clinical conducted after 2–3  days and is valid for 7  days. The
manifestations of oral TB are varied and usually manifests evaluation is based on the diameter of the inflammation
as nonhealing ulcer but can also appear as nodules, area measured transversally against the longitudinal
swelling, fissures and as osteomyelitis of jaw bones. Oral direction of the challenged forearm. An inflammation area
TB ulcers are usually single rather than multiple; they over 10 mm in immunocompetent subjects is considered a
have an indurated, irregular and undermined margin positive result. In immunocompromised patients, an area
with a necrotic base. The ulcer may be initially painless larger than 5 mm indicates TB. In turn, the minimum size
but may become painful with the passage of time.[20] of inflammatory area in low‑risk individuals and children
Other manifestations of oral TB may include nodular under 15 years of age is 15 mm. Although the Mantoux
lesion or cold abscess.[6,19] Three forms of oral TB have reaction is the method of choice in TB diagnosis, the
been described: acute miliary, chronic ulcerative and test has a few limitations, such as the low sensitivity in
lupus vulgaris.[21] Overwhelming majority (about 93%) immunocompromised patients (which points to the risk
of the oral lesions are ulcers and approximately half of false negative results), the difficulty to use in children,
336 Journal of Oral and Maxillofacial Pathology | Volume 24 | Issue 2 | May-August 2020
Hamid, et al.: Primary oral tuberculosis – A case series of a rare disease

the subjective character of interpretations and the need not be published and due efforts will be made to conceal
for a second appointment for confirmation purposes in their identity, but anonymity cannot be guaranteed.
some cases.[30] The oral manifestation of TB may present as
an ulcerative, painless lesion on the palate, lips or tongue, Financial support and sponsorship
accompanied by persistent cervical lymphadenopathy.[29] Nil.
Our first case showed non painful ulcer on gingiva, second
Conflicts of interest
one had painful ulcer on palate, third case had non painful
gingival swelling and the fourth one had painful ulcer There are no conflicts of interest.
on tongue. The male:female ratio in our series was 1:3. REFERENCES
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338 Journal of Oral and Maxillofacial Pathology | Volume 24 | Issue 2 | May-August 2020

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